Dyskinesia-hyperpyrexia syndrome in a patient with Parkinson’s disease: a case report

Abstract

Non-physiological, excessive dopaminergic stimulation can cause dyskinesia-hyperpyrexia syndrome (DHS), which was initially reported by Gil-Navarro and Grandas in 2010. A 70-years-old woman with a 13-years history of Parkinson's disease (PD) was hospitalized due to difficulty walking, despite being treated with levodopa/carbidopa (600 mg/day), immediate-release pramipexole (3 mg/day), and selegiline (5 mg/day). Immediate-release pramipexole was changed to extended-release pramipexole without changing the dose or levodopa equivalent dose (LED). The patient's adherence to drugs was good. The parkinsonism gradually improved and the patient was discharged. One month later, the patient developed severe generalized athetotic dyskinesia with visual hallucinations and hyperpyrexia that lasted for a week, and she was readmitted to hospital. On admission, the patient was conscious but slightly disoriented. Body temperature was 40.3°C with hyperhidrosis. Leukocyte count in the peripheral blood was 1.78×10(4)/ml and serum creatine kinase was >3×10(4) U/l. Chest survey, whole-body computed tomography, and cranial magnetic resonance imaging showed no abnormalities. The patient was diagnosed with DHS and treated by tapering the oral administration of dopaminergic drugs, including extended-release pramipexole. Her clinical condition recovered without dyskinesia, and serum creatine kinase level swiftly normalized. DHS and resemblant conditions are reported to occur in long-term PD patients with motor complications. In advanced stage PD, loss of dopaminergic neurons impairs the dopamine holding capacity of the striatum and exogenous dopaminergic drugs can result in uncontrollable and excessive fluctuations in dopamine concentration. Our case recommends caution when switching to long-acting dopaminergic drugs, even if the dose is unchanged, could lead to excessive dopaminergic stimulation. This case highlights the importance of considering both the LED and the duration of action of dopaminergic drugs when adjusting medication.

Dyskinesia‐Hyperpyrexia Syndrome in Parkinson's Disease: A Heat Shock–Related Emergency?

“…DHS is a poorly described and under‐reported acute complication in PD patients with an opposite clinical spectrum (dyskinesia), but under similar precipitating systemic factors (infectious diseases, trauma, and dehydration). In all reported cases, including ours (Table , n = 8 patients), DHS occurred in patients with long duration PD and was associated with a trigger event. Most of the subjects were treated with high doses of dopaminergic drugs (half of them with LCIG).…”

“…Nonphysiological dopaminergic stimulation can cause Dyskinesia‐Hyperpyrexia syndrome (DHS), a rare medical emergency associated with Parkinson's Disease (PD) . DHS is characterized by severe continuous dyskinesia, rhabdomyolysis, and hyperthermia that may progress to mental state alteration, renal and cardiac failure, and death.…”

Summertime Dyskinesia-Hyperpyrexia Syndrome: The “Dual Heat” Hypothesis

“…Nonetheless, this hypothesis is insufficient to explain the incapacitating dyskinesia in our case and other cases. 1,2 Despite the anecdotal nature of this case, the excellent response to an enteral L-dopa infusion allowed us to speculate a differential pathogenic mechanism and eventually report a mean for treating refractory dyskinesia-hyperpyrexia. Further studies are needed to support these findings and clarify the impairment of thermoregulatory pathways in Parkinson disease.…”

“…1,2 Here, we present another case that unexpectedly resolved after receiving an enteral infusion of L-dopa. Unlike current theories, we hypothesize a central nervous system origin for this syndrome.…”

Levodopa-carbidopa enteral suspension in advanced Parkinson’s disease: clinical evidence and experience

“…The dyskinesia-hyperpyrexia syndrome, comprising severe dyskinesia, hyperthermia, rhabdomyolysis and a confusional state, is a rare complication of PD treatment that may be associated with a high level of dopaminergic stimulation [GilNavarro and Grandas, 2010;Taguchi et al 2015]. In a case study of a PD patient who had experienced episodes of dyskinesia-hyperpyrexia during three consecutive summers while receiving oral levodopa, commencement of LCIG therapy led to a complete cessation of further episodes [HerrerosRodriguez and Sánchez-Ferro, 2016].…”